RESUMO
Tubulointerstitial nephritis and uveitis (TINU) syndrome is defined as the occurrence of tubulointerstitial nephritis and uveitis in the absence of other systemic diseases. Three pediatric cases have been reported in the Republic of Korea, and we now report a fourth case. A 15-year-old girl presented to the ophthalmology department with a 1-week history of bilateral ocular discomfort that worsened on the day of presentation with redness and pain in both eyes. She was diagnosed with bilateral uveitis, and her baseline examination revealed moderate renal dysfunction and mild proteinuria. A renal biopsy was performed and confirmed the diagnosis of TINU syndrome. She was started on steroid eye drops and a 12-week course of oral steroids at a dose of 40 mg/m2/day, which completely resolved the proteinuria and mild renal function to an estimated glomerular filtration rate of 60 mL/min/1.73 m2. However, the uveitis did not improve, and despite the addition of oral methotrexate as a second-line treatment, the uveitis remains unresponsive to treatment over 21 months. Further evaluation and treatment are ongoing, and active therapeutic intervention is suggested even at a pediatric age, considering the lack of improvement in renal function and uveitis to date.
RESUMO
Pagonimus Westmani, occuring as an important human infestation of the lung, is widely distributed in the Far East ; Korea, Japan, China and the Philippines. Since Otani found the parasites for the first time in the human brain in 1887, it has been established that cerebral infestation occurs with the highest frequency among extrapulmonary involvement. In 0.8 to 25 percent of patients infested with the lung flukes cerebral paragonimiasis may be developed. The larvae may procedure arachnoiditis, abscess, fibrous cystic lesions and granulomas in the brain. Based on a review of the literatures and upon the 7 cases of cerebral paragonimiasis which have been observed by the authors at the National Medical Center, Seoul, Korea, the following points are stressed. 1) Males had a significantly higher prevalence than females. Cerebral paragonimiasis was most commonly found in young adults(16 to 25yrs). 2) Clinically the patients with cerebral involvement were characterized by Jacksonian type of epilepsy, headache and visual disturbance. Mental deterioration, hemiplegia, hemihypesthesia, homonymous hemianopsia and optic atrophy were the five major signs. 3) The patients having symptoms for less than 6 months developed mild leukocytosis in many cases. All the cases of cerebral paragonimiasis revealed positive reaction in the intradermal tests with purified antigens of P. Westermani. 4) On skull films multiple round or oval cystic calcification was diagnostic value for cerebral paragonimiasis. On angiogram vascular staining and abnormal vessels were not disclosed. On pneumoencephalogram subcortical atrophy or filling defect of the ventricle may be showed. 5) The predilection area of the granuloma and fibrous cystic mass were the occipital, posterior parietal and posterior temporal lobes. 6) The patients with chronic stabilized cerebral paragonimiasis. having intractable symptoms and signs, could not be improved with Bithionol treatment. But headache, vomiting, visual disturbance, facial weakness and especially intractable seizure were improved in our cases treated with surgical operation.